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PRIMARY ORBITAL LIPOSARCOMA: A CASE REPORT
Authors: Chebbi A1, Hachicha F1, Ben Hssine L�, Feki O1, Bouayed E1, Malek I1, Zeghal I1, Bellagha I � , Bouguila H, Nacef L1.
1 Ophthalmology Department, Hedi Raeis.
� Radiology Department, Children Hospital.
University Tunis El Manar, Faculty of medicine of Tunis, Tunisia. 
Introduction:
Primary liposarcoma is a malignant soft tissue tumor which usually originates from mature adipose tissue and is most common in thigh and retroperitoneum [1]. Although liposarcoma represents the most common soft tissue sarcoma in adulthood, primary and secondary liposarcoma of the orbit are rare [2,3]. In part because of the rarity of such tumors, definitive initial histopathologic diagnosis is often difficult [4].
We describe a new case of primary orbital liposarcoma.
Case report:
A 40 year-old female, with no past medical history, presented with right axial progressive painless non-pulsatile and non-inflammatory proptosis associated with a reduction in visual acuity (Figure 1). Her best corrected vision was 5/10 P2 in the right eye and 10/10 P2 in the left eye. Anterior segment examination was unremarkable with no relative afferent pupillary defect, intraocular pressure and eye fundus was normal bilaterally. Ocular motility was not restricted. General examination was normal. A Goldmann perimetry revealed constricted visual fields.
MRI (Figure 2) revealed an expansive intraconal mass with well-defined borders involving the optic nerve. T1-weighted images showed an hypo signal mass, on T2 and after Gadolinium contrast, the mass showed a hyperintense signal. There was no destruction of the bone wall.
 As presence of compression signs, surgical excision was performed and revealed a mass of hard consistency with greyich-yellow surface. Histological examination revealed proliferating spindle-shaped and�stellate�cells with�lipoblasts�suspended within�myxoid-rich matrix and rich vascular pattern. The appearances were typical of a Myxoid�liposarcoma (Figure 3).
Few months later, she developed recurrent painful proptosis with pain when moving the globe. The MRI revealed an ill-defined extraconal mass that was displacing the eyeball inwards. T1-weighted showed a hypo signal mass, on T2 and after Gadolinium contrast, the mass showed a hyperintense signal (Figure 4). The diagnosis of recurrent liposarcoma was made and surgical revision with adjuvant radiotherapy was performed. A total dose of 60 Gy was administered at 18 Gy/session, and was well tolerated. She was followed up for 12 months. There was no evidence of recurrence. On the last follow up examination, there was no proptosis of right eye. Her visual acuity was the same result as pre-operation, intraocular pressure was normal bilaterally. Both optic discs were normal. There was no evidence of the radiation neither retinopathy nor radiation optic neuropathy.
Discussion:
Liposarcoma is rare, it represents 10 � 20% of all sarcomas. [2].The first description of orbital liposarcoma by Strauss appeared in 1911 [5]. Despite the fat richness of the orbit, primitive liposarcoma is rare with only 40 cases reported in the literature contrary to secondary liposarcoma [5,7]. Orbital liposarcoma with metastasis are even rare with only 6 cases reported [8].
Liposarcoma is more frequent in men than in women and is detected at an average patient age of 53 years. Compared with nonorbital liposarcoma, orbital liposarcoma has been shown to have a predilection to occur in younger patients [6].
The development of an orbital liposarcoma is often accompanied by clinical signs secondary to compression of orbital structures, the main symptoms are proptosis (92%), diplopia (42%), reduction in visual acuity (29%) and ptosis [1,5,6]. The duration of the symptoms before presentation ranges from 2 weeks to 7 years [9]. Radiological investigations including CT and MRI are essential to visualize the tumor and evaluate its local extension.
The tumor originates from primitive stromal cells related to intermuscular fascial planes or from perivascular pluripotential mesenchymal cells [6]. World Health Organization (WHO) recognizes five histologic types of liposarcoma: well differentiated, myxoid, round cell, pleomorphic and dedifferentiated [10]. The link between the various types of liposarcomas is the lipoblast [6]. Myxoid liposarcoma is the most common histological type (55%) [5], the larger proportion of myxoid types in the orbit correlates well with liposarcoma found in the soft tissues in general, where it is thought to account for 40% to 50% of all liposarcoma subtypes [6].
The clinical differential diagnosis of orbital liposarcoma includes any orbital mass with non-distinctive radiologic appearance [6]. The histological differential diagnosis of orbital liposarcoma is depending on the histologic type. The well-differentiated type may mimic normal adipose, main differences are the presence of variation in fat cell size and atypical hyperchromatic cells. Lipoblastic meningiomas with signet-ring cells may mimic liposarcoma but the vacuolated appearance of a lipoblastic meningioma is more prominent than in a true liposarcoma [2]. Myxoid pleomorphic lipoma can also mimic liposarcoma but tumor circumscription, presence of ropy collagen bundles, and lack of lipoblasts advocate myxoid pleomorphic lipoma [6].
Sometimes initial histologic diagnosis is uncertain and modern immunohistochemical techniques using antibodies to S-100 protein, CD34, smooth muscle actin, and desmin may increase diagnostic precision [6].
The prognosis depends on several factors: size and location, histological type and grade of differentiation; Well differentiated and myxoid liposarcomas have a relatively indolent course, whereas the round cell and pleomorphic variants usually behave aggressively with more distant metastasis. Deaths from soft tissue liposarcoma in general are often as a result of the local effects of tumors and the oft inability to achieve clear resection margins because of disease location [4, 6].
Treatment of orbital liposarcoma remains controversial with some surgeons performing repeated local resections for recurrence and others performing an initial exenteration after histologic confirmation of disease [6]. Indeed surgical excision with ample margins admittedly improves survival and local control [11,12], but most of the orbital liposarcomas cannot be completely excised without causing damage to infiltrated structures, particularly the extraocular muscles [6]. Association of radiotherapy provided best results [13]. Myxoid liposarcomas are believed to be radiosensitive if not radiocurable but well-differentiated tumors are less responsive [6]. Conservative surgery is indicated for small, well defined and easily accessible tumors, with adjuvant radiotherapy, while exenteration is recommended for invasive, dedifferentiated and recurrent tumors [6, 11].
The use of chemotherapy is controversial, it has been reported that Dexorubicine and Dacarbazine are effective in the treatment of myxoid liposarcoma [6, 14].
Conclusion:
The primary orbital liposarcoma is a rare entity; the positive diagnosis is based on imaging and confirmed by histological study after excision biopsy, diagnosis may be difficult because of initial histopathologic uncertainty. Exenteration after confirmatory biopsy seems to provide the best chance of a cure, with increasing evidence for the use of adjuvant radiotherapy. Prognosis and management depend on several factors including the size and histological type.
References:
1-Lane CM, W right JE, Olmer A (1988). Primary myxoid  liposarcoma of the orbit. Br J Ophthalmol 72: 912�917.
2-Cai YC, Mc Menamin ME, Rose G, Sandy Charles J, Cree Ian A, et al (2001). Primary liposarcoma of the orbit: a clinicopathologic study of seven cases. Ann Diagn Pathol 5:255� 66.
3-Rosner M, Yosepovich A, Paul M, N Rosen, M Perelman (2006).  Orbital well differentiated liposarcoma demonstrating chromosomal imbalances. Eye 20:126�8.
4-Simon N. Madge, Krishna Tumuluri, Diego Strianese, Paola Bonavolonta, Geoff Wilcsek et al (2010). Primary Orbital Liposarcoma. Ophthalmology 117:606�614.
5-Jing-Xue Zhang, Jian-Min Ma, Ning-Li Wang (2011). Dedifferentiated orbital liposarcoma: a case report. HYPERLINK "http://www.ncbi.nlm.nih.gov/pubmed/22553700" \o "International journal of ophthalmology."Int J Ophthalmology 4:452-3.
6-Asit Ranjan Mridha, Mehar Chand Sharma, Chitra Sarkar, Vaishali Suri, Ajay Garg, et al. (2007). Primary liposarcoma of the orbit: a report of two cases. Can J Ophtalmology 42: 481-483.
7-STELA GIURI, M. RAICA, M. MUNTEANU (2013). Myxoid/round cell conjunctival liposarcoma. A case report. Rom J Morphol Embryol  54(3):655�658.
 8-Hannachi S, Braham E, Bhouri L, K Mrad, I Abbes, et al (2007). Orbital metastasis of liposarcoma.  Fr J of Ophtalmology 30: e28.
9-Madeline Doyle, Alexandre N. Odashiro, Patr�cia R. Pereira, Macanori Odashiro,
Fernando Kobayashi and al. (2012) Primary Pleomorphic Liposarcoma of the Orbit: A Case Report. Orbit, 31(3): 168�170.
10- Enterline HT, Culberson JD, Rochlin DB, Brady W. (1960) A clinical and pathological study of 53 cases. Cancer 13: 930-50
11-A. Costas, P. Castro, J. M. Munuoz (2001) Primary orbital liposarcoma: case report and review of the literature. Int. J Oral Maxillofacial Surgeons 30: 170�172.
12-Krauss DH, Dubner S, Harrison LB, Strong EW, Hadju SI and al. (1994) Pronostic factors for recurrence and survival in head and neck soft tissue sarcomas. Cancer 74: 697�702.
13-SABB PC, SYED NA, SIRES BS, LEMKE BN, DORTZBACH RK and al. (1996) Primary orbital myxoid liposarcoma presenting as orbital  pain. Arch Ophthalmology 114: 353�354.
14-Patel SR, Burgess MA, Plager C, Papadopoulos NE, Linke KA and al. (1994) Myxoid liposarcoma: experience with chemotherapy. Cancer 74:1265�9.



FIGURE LEGENDS
Figure 1: Right axial progressive painless non-pulsatile and non-inflammatory proptosis 
Figue 2: MRI showing large retro-ocular and intraconal mass, developed near the apex of the right orbit with a low signal on T1 weighted images and iso signal on T2. After Gadolinium contrast, the mass showed an hyper intense signal, a compression of the right optic nerve without anomalies of orbital fat or extraocular eye muscles.
Figure 3: Figure 3: histological study, Proliferating spindle-shaped and�stellate�cells with�lipoblasts�suspended within�myxoid-rich matrix and rich vascular  patter (H and E, x400): Myxoid�liposarcoma.
Figure 4: MRI showing local recurrence: a retro-ocular, extraconal and intraconal mass in the right orbit, infiltrating superior and lateral rectus muscles, with iso signal on T1 weighted images, a slight signal on T2 weighted images and an hyper intense signal after Gadolinium contrast. 



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